Objective: To describe a case of intracytoplasmic sperm injection (ICSI) with testicular sperm in men with immotile cilia syndrome and to discuss the role of micromanipulation in the treatment of these patients. Design: Case report. Setting: Private infertility clinic and assisted reproduction unit. Patient(s): Couple with male factor infertility due to Kartagener's/immotile cilia syndrome. Intervention(s): The patient's partner underwent ICSI with testicular sperm. Main Outcome Measure(s): Semen characteristics, sperm viability, fertilization and cleavage rate, pregnancy, and birth after ICSI. Result(s): With testicular sperm, the two pronuclear fertilization rates were 53%. ICSI was successful in the first cycle. The uncomplicated pregnancy resulted in the birth of two healthy children, male/female twins. Conclusion(s): With testicular sperm, successful oocyte fertilization after ICSI in couples with male Kartagener or immotile cilia syndrome is possible with nonprogressive motile sperm resulting in clinically healthy offsprings. (Fertil Steril (R) 2007; 88: 497.e9-11. (C) 2007 by American Society for Reproductive Medicine.)